Literature DB >> 29182795

Epidermolysis Bullosa (EB) Acquisita in an Adult Patient with Previously Unrecognized Mild Dystrophic EB and Biallelic COL7A1 Mutations.

Liliana Guerra1, Angelo Giuseppe Condorelli, Paola Fortugno, Valentina Calabresi, Cristina Pedicelli, Giovanni Di Zenzo, Daniele Castiglia.   

Abstract

Circulating anti-type VII collagen autoantibodies are frequently detected in patients with recessive dystrophic epidermolysis bullosa (RDEB). However, evidence supporting their pathogenic role in inducing epidermolysis bullosa acquisita (EBA) has been provided for only one individual with dominant dystrophic epidermolysis bullosa (DDEB). We describe here a patient who presented with dystrophic toenails since early childhood and developed trauma-induced skin blisters and oral erosions at age 26 years. Direct immunofluorescence showed IgG deposits with a u-serrated pattern along the cutaneous basement membrane zone, while no change in the expression of collagen VII could be detected by antigen mapping. High-titre anti-collagen VII antibodies were detected by enzyme-linked immunoassay (ELISA). In parallel, sequencing of epidermolysis bullosa (EB) genes identified compound heterozygous COL7A1 missense c.410G>A (p.Arg137Gln) and splicing c.3674C>T (p.Ala1225_Gln1241del) mutations, previously unrecognized in dystrophic epidermolysis bullosa (DEB). Thus, our patient had RDEB "nails-only" and developed mechanobullous EBA in adulthood. These data support a pathogenic role of circulating autoantibodies to collagen VII in inducing EBA in selected patients with DEB. Unforeseen worsening of skin symptoms in DEB should prompt laboratory investigations for EBA.

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Year:  2018        PMID: 29182795     DOI: 10.2340/00015555-2851

Source DB:  PubMed          Journal:  Acta Derm Venereol        ISSN: 0001-5555            Impact factor:   4.437


  5 in total

Review 1.  The Immunogenetics of Autoimmune Blistering Diseases.

Authors:  Diana Kneiber; Eric H Kowalski; Kyle T Amber
Journal:  Adv Exp Med Biol       Date:  2022       Impact factor: 2.622

2.  Case Report: Diagnostic and Therapeutic Challenges in Severe Mechanobullous Epidermolysis Bullosa Acquisita.

Authors:  Franziska Schauer; Alexander Nyström; Manfred Kunz; Stefanie Hübner; Sarah Scholl; Ioannis Athanasiou; Svenja Alter; Judith Fischer; Cristina Has; Dimitra Kiritsi
Journal:  Front Immunol       Date:  2022-04-07       Impact factor: 8.786

3.  Case report: bullous pemphigoid development underlies dystrophic epidermolysis bullosa disease worsening.

Authors:  Giovanni Di Zenzo; Giovanna Floriddia; Sabrina Rossi; Feliciana Mariotti; Alessia Primerano; Angelo Giuseppe Condorelli; Biagio Didona; Daniele Castiglia
Journal:  Front Immunol       Date:  2022-07-29       Impact factor: 8.786

4.  Occurrence of autoantibodies against skin proteins in patients with hereditary epidermolysis bullosa predisposes to development of autoimmune blistering disease.

Authors:  Saskia Lehr; Felicitas Felber; Iliana Tantcheva-Poór; Christina Keßler; Rüdiger Eming; Alexander Nyström; Marta Rizzi; Dimitra Kiritsi
Journal:  Front Immunol       Date:  2022-07-25       Impact factor: 8.786

Review 5.  A Review of Acquired Autoimmune Blistering Diseases in Inherited Epidermolysis Bullosa: Implications for the Future of Gene Therapy.

Authors:  Payal M Patel; Virginia A Jones; Christy T Behnam; Giovanni Di Zenzo; Kyle T Amber
Journal:  Antibodies (Basel)       Date:  2021-05-17
  5 in total

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